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Families of children with Down syndrome

Cliff Cunningham

The paper presents an overview, from a personal perspective, of the key research findings of the longitudinal study of the Manchester Down Syndrome Cohort. The study began in 1973 and is currently visiting the families as the young people enter adulthood. At present over 100 families remain in the cohort and provide a representative sample of families of children with Down syndrome in the UK during these years. It is one of the largest and most detailed multifactorial studies in the field of Down syndrome. The overriding impression of the families and their child with Down syndrome is one of normality. The factors that influence the well being of all members are largely the same as those influencing any child or family. The majority of families do not exhibit pathology as a consequence of having a child with Down syndrome. Indeed the evidence points to positive effects for many families when one member has Down syndrome. The results also emphasise the diversity of families and of individuals with Down syndrome. Some families and children with Down syndrome are vulnerable and at risk. The research has begun to identify who these may be and suggest possible directions for more effective support and intervention.

Cunningham CC. Families of children with Down syndrome. Down Syndrome Research and Practice. 1996;4(3);87-95.

Introduction

This paper focuses on two key questions:

What is the effect of having a child with Down syndrome on the family?
- How does the child effect the way they function as a system? What factors
put them at risk and conversely, what factors provide resilience to potential
stressors?

What is the effect of the family on the child? - in other words what
factors positively or negatively influence the development and well-being
of the child?

I will be mainly using the research results, coloured by my own experiences,
from the Manchester Down syndrome Cohort Study which I started in 1973.
I have been fortunate in being able to follow over 100 families over the
years and the last project, completed by my colleagues
Sloper and Turner
(1994) investigated them in the teenage years. Currently we are visiting
them again as they enter adulthood.

I will begin by briefly describing the cohort and the families, and the
background to the various studies we have made.

Background

In order to give an overview of the key findings I will have to leave out
detailed descriptions of methodology. These can be found in the references
listed. We have used an extensive range of measures based on both qualitative
and quantitative methods. All the research reported has been subject to
critical peer review and published in refereed journals or project reports.

I would like to emphasise that we view the family as a transactional system
in which many variables are constantly interacting on each other and changing.
To cope with this dynamic system we have used multivariate approaches in
an attempt to identify the most influential factors associated with stability
and change over time that influence family well-being. But beyond this we
are also seeking to describe a style of how families deal with life circumstances
that increases or decreases the chances of well being (Rutter
1989).

If one is to do this it is essential that the sample is representative of
the population of families of children with Down syndrome.

The cohort

Between 1973 and 1980 we were notified of 203 births. Twenty-two families
did not join the project because the babies died, were placed out of the
area in care or parents moved or did not reply to our invitation. One hundred
and eighty one families joined the project which represents between 85 and
90% of all births of infants with Down syndrome in the area during this
period.

The families did not differ from similar families in Britain at this time
with the exception that there was a small bias toward more non-manual and
more educated and financially better-off families. This has been found in
other cohorts of families of children with Down syndrome ( e.g.
Carr 1988 ). Also,
the average age of parents was slightly older as would be expected given
the increased incidence of births to older mothers. Thus the cohort included
some teenage mothers and single parent families, families from ethnic minorities
and second marriages. Some were rich and some were very poor. Some had large
extended families and some small nuclear families.

This is to be expected as the incidence of Down syndrome is largely the
same across different races and cultures. The point to be emphasised is
that the families with a child with Down syndrome are no different to other
families when the child is born and exhibit the same large individual differences.
There is not, therefore, a typical type of family of a child with Down syndrome.

The children with Down syndrome also show a wide range of individual differences
in terms of their cognitive, social and physical abilities, health status
and personality. For example we found IQ scores in the pre-school years
ranged from less than 20 to 100 with a mean around 50.
Carr (1988) reported
similar ranges in the early years and when she followed up her cohort at
21 years of age the IQ's ranged from less than 20 to 70, with a mean of
40. It is worth noting that this range covers some 60 IQ points which is
similar to the variance found for the 'normal' population. We also found
a wide range of differences in temperament in the early childhood years
(Gibbs 1984) similar
to that found in normal distributions but with more children showing a dampened
and passive personality and over 70% being relatively easy to manage and
interact with. However 25 to 30% had difficult temperaments, tended to be
restless and excitable and were difficult to manage.

A problem with longitudinal studies is that individuals are lost over time.
In our cohort we had 134 families when the children were five years old.
The attrition during this period was due to deaths and families moving away.
No parent had dropped out of the study. In our last study in 1991 we had
contact with 118 families and 106 took part in the study. The fall in numbers
was again largely due to families moving and some deaths. However several
have refused to take part in later studies for many reasons. Therefore we
carefully examined how representative the current cohort is compared with
our original group at each follow-up. Over the years the proportion of single
parent families has increased from 4 to 15% which compares to 20% in the
general population. We also have fewer mothers in employment compared with
the general population - a finding reported in many studies of children
with disability. So far we have not found any significant differences on
major variables for the follow-up studies and feel reasonably confident
that we have representative findings.

The studies

There have been five major studies over the years. The first was the early
development and intervention study (1973-1982). We visited all families
at least every six weeks and gave them structured programmes to stimulate
their infant and discussed any problems. We collected demographic data,
developmental assessments of the child and descriptive information of the
children's health and problems experienced by the families. The second study
(1982) obtained mother's perceptions about family adaptation and functioning
using interviews and we measured maternal stress and child behaviour problems.
In 1984-85, the third study examined the long-term effects of the early
intervention and behaviour problems in the cohort compared to a comparison
group (these studies are reported in detail in
Byrne et al 1988,
Cunningham 1987).
The fourth study in 1986-88 was a much more detailed multivariate study
of family adaptation which included information from fathers and was repeated
five years later in 1991 when the children were in their teenage years.
This study also included information from the siblings for the first time.

Theoretical focus

Early research on families of children with disability reflected a pathological
model in that families were automatically assumed to suffer as a consequence
of the child - in effect the studies only looked for negative outcomes (Byrne
and Cunningham 1985). We adopted a transactional family system model
and combined this with the
Folkman and Lazerus (1985) cognitive theory of
stress and coping. This approach argues that any event, such as the disability
of a family member, will have different meanings and effects for each individual
and that only the individual can appraise whether the effect is distressful.
The potential effect of such an event will be modified by the availability
and use of resources and coping strategies. Resources include:

physical - health, energy and stamina,

utilitarian - finances, housing, employment,

social - social networks and support systems,

psychological - belief systems, problem solving skills,
personality.

Lack of resources to cope with a situation are likely to increase strain
or stress on the family situation. Hence they constitute a need (Cunningham
and Davis 1985). Coping strategies refer to how parents try to deal
with problems and reduce the strain or stress they cause. They can be focused
on emotion or practical problems. Strategies range from passive e.g. wishful
thinking (hoping the problem will go away), stoical (making light of the
problem), through to active strategies e.g. problem solving. If the stressful
situation is resolved the person is more likely to feel strong and in control,
which can then strengthen their coping resources for the future. If not
resolved this places a chronic burden on resources and may lead to negative
effects on physiological and psychological well-being.

Thus our approach has been to assess an extensive range of factors that
might have negative or positive effects on the family and child with Down
syndrome.

Child measures include: mental ability using developmental and IQ tests,
self-sufficiency indicating degree of personal, domestic and social
independent living skills, social life encompassing friendships
and use of leisure time, behaviour problems, and academic attainments.
These are the main outcome measures but we also measured a number of descriptor
variables: gender, age, position in family, health problems, temperament
on the dimension of excitability-distractibility, supervision and caretaking
needs, intensity of early intervention and type of school attended.

The two main outcome measures used to assess parents were distress,
using a 24 item checklist of psychosomatic symptoms, and satisfaction
with life, based on an index of how mothers and fathers felt about
their life in general, social support, time for self, life with partner
and other family members, finances and parenting. Parents also completed
a number of validated scales about their personality, locus of control,
marital relationships, social support, contact with services and perceived
satisfaction with services. To investigate family functioning, parents completed
the Family Environment Scale (Moos
and Moos 1981) and the Family Relations Index (Holahan
and Moos 1983). Parent-child relationships and attachment were assessed
from interview. In addition they also completed a Life-events scale and
the ways of Coping Questionnaire (
Folkman and Lazerus 1985) adapted for the families of children with
Down syndrome (Knussen
et al 1992). The demographic variables measured were social
class, unemployment, parent educational level, housing, car ownership, marital
status, age of parent, size of family, financial problems.

What are the effects of having a child with Down syndrome on the
family

The weight of evidence from all the studies is that the majority of the
families (65-70%) function like most families and are not 'at risk' because
of the child's Down syndrome. They did not show above thresholds of psychological
and physiological distress and the scores were lower when compared with
studies of families of children with other disabilities (Quine
and Pahl 1989,
Sloper
and Turner 1993). On the family functioning measures they exhibited
good internal relationships, cohesion and expressiveness.
Mink et al. (1983)
reported that 68% of their families with children with Down syndrome were
rated as cohesive and harmonious and had significantly higher morale than
families of children with other intellectual disabilities. Three-quarters
of our families were consistently rated over the years for positive expressions
of warmth and affection toward their child with Down syndrome. The divorce
rate in the cohort has always been lower than the national average and only
14% felt that having a child with Down syndrome had had a detrimental effect
on their marriage. Hence the majority of families report good marital relationships.
This has also been recently reported for an Australian sample (Cuskelly
and Dodds 1992). In 1984 and 1991 we asked mothers about the effects
of having a child with Down syndrome on themselves and the family. Less
than a third thought there had been detrimental effects. The majority felt
there were no real detrimental effects and usually responded that they had
changed for the better. Typically they felt they were less concerned with
trivia, less materialistic and self-centred. Their comments indicate a shift
in beliefs to more pro-social behaviour. They also felt their partners and
other children had benefited in the same way.
Mink et al. (1983) speculated
that the higher morale in families of children with Down syndrome could
be because of a positive effect of the child on the home climate.

Thus the picture, in childhood, is that the child with Down syndrome is
not a burden and mere recipient of family care for most families. In fact
they appear to make a positive contribution.

By the teenage years, a decrease was found in mothers' perceived satisfaction
with life. This was associated with a decline in actual and perceived satisfaction
with social support. There was also a trend for more mothers to perceive
negative effects of the child with Down syndrome on the family. It would
appear that they felt their teenager with Down syndrome was placing greater
restrictions on family life compared to a non-disabled teenager. In the
'normal' family life-cycle most parents experience more independence as
the children reach the teenage years and become more independent. But this
does not happen for the majority of teenagers with Down syndrome. Their
slower development and learning disabilities forces them to get out of step
with the normal life cycle. Depending on their level of ability, skills
and interests, they begin to have fewer social contacts and friends who
are not disabled. By the teenage years many are relatively isolated and
become increasingly dependent on the family for social interaction. As one
father succinctly described it "I have become the entertainment organiser
for my son". The greater the supervision needs of the child the greater
the restrictions on the family. For example, we found that the level of
behaviour problems in the children was significantly related to reduced
social contacts, friendships and increased ratings of negative effects on
the family.

Potential risk factors

Between a quarter and a third of the families were experiencing difficulties.
This was reflected in higher levels of measured distress and lower satisfaction
with life. The longitudinal analysis showed that the strongest predictor
of any measure in the teenage years was the score on that measure five years
earlier. For example, the measure of stress for mothers was particularly
stable over the 9 years from early childhood to the teenage.

This is important as it indicates that the pattern of family and child functioning
becomes relatively well established in the childhood years. The implication
is that any intervention to change the pattern must focus on the early years.

For mothers and fathers the factor most associated with negative stress
was behaviour problems in the child. For the majority of families the management
of the child became easier as they got older and this was associated with
increases in self-sufficiency and reduced behaviour problems. Despite this,
the level of behaviour problems found in the early childhood years strongly
predicted later levels and the scores became increasingly stable from the
mid-childhood to the teenage years. The children most likely to have significant
behaviour problems had lower mental ability and suffered from repeated infections
and health problems in the childhood years. Studies have shown that behaviour
problems are more likely in children with prolonged hospitalisation, chronic
ill-health and injury. The suggestion is that such events increase strain
on the family and can also alter child management methods and expectations
of child functioning. This can lead to increased over-protection by parents
and increased dependency for the children. If this occurs in families with
relationship problems the effect is compounded. We found that behaviour
problems in the sibling were significantly associated with poor family relationship
patterns but not with any characteristics of the child with Down syndrome.

Fathers of children with low IQ, with or without behaviour problems, and
more so if the child was a boy, consistently reported lower satisfaction
with life over the years. This supports the suggestion, commonly reported
in the literature and by mothers, that fathers are more likely to have problems
of adaptation. The recent longitudinal study over the first years of life
by
Shonkoff et al.
(1992) found that fathers continually reported higher
levels of stress than mothers in their attachment to the child and that
these levels were higher for fathers of children with Down syndrome compared
to other disabilities. Thus many fathers may find they are less fulfilled
in their parenting role because their child has Down syndrome. This may
reflect their problems in coming to terms with having such a child.

It is possible, however, that these increased problems are not just about
male pride and ego. Early intervention efforts frequently focus on mothers
because fathers are often at work during contact time. Thus fathers receive
less support. Hence we must make efforts to meet fathers individual needs.
In my experience this is not easy as many fathers are reluctant to attend
meetings or seek counselling. However in the weeks following the birth many
parents are in a high state of uncertainty and are actively seeking guidance.
I find this an opportune time to establish relationships which, if they
are perceived by mothers and fathers to be helpful, can last over the years.
Within this, one has an opportunity to overcome male resistance and possibly
help with problems of adaptation. But this has to be a well formulated individual
approach.

Thus low functioning and behaviour problems which occur in less than a third
of the children with Down syndrome is a risk factor for family well-being.
However even when the child is functioning at this level it is not inevitable
that the families experience the same levels of stress or low satisfaction.
The effects of these characteristics on well being are modified by the family
resources. Our results show three important groups: social-economic, personality
and family relationship variables.

Family well-being was associated with lack of utilitarian resources, rather
than social class status. For example inadequate housing predicted levels
of behaviour problems and through this parental stress. Financial problems
and lack of a car was associated with mothers' stress levels and poorer
family relationships. If both parents were employed mothers reported less
strain and more satisfaction. Those mothers in employment had lower stress
scores than those who were unemployed and this was independent of previous
stress scores. Thus it appears that by the later childhood and teenage years
employment outside the home had some protective effect for mothers' well
being. This may be because they have more social contacts and access to
support, respite from the demands of the home and child, alternative interests
and the chance to appraise their situation with people outside of the family.

Unfortunately, compared with national figures, the mothers of children with
disabilities are less likely to be in employment. Presumably this is because
of the care needs of their child. However we found in the early studies
those from our cohort were more likely to be employed than a comparison
group. We felt this was a result of the support and encouragement we gave
to seek employment rather than just feeling they had to be with the child
because of the disability. Even so, 40% of the mothers in the later studies
stated that the child's disability restricted their own employment opportunities.
This was often related to the need for after school and holiday care which
for some children needed to be specialized. Given the potential effect
of mother employment on family finances, housing and general well being,
and through this the child's development and quality of life, the provision
of services assisting their employment would appear to be a worthwhile endeavour.

The personality of parents was measured on the dimension of neuroticism.
The number having relatively high levels was no greater than expected for
the general population. Detail analysis indicated that high neuroticism
in parents indicated a personal vulnerability to stressors and higher stress
scores. Higher neuroticism scores were also associated with higher levels
of child behaviour problems. However when the parent had the resources of
social support and used practical problem solving strategies to cope, the
potential effects of neuroticism on levels of stress and child functioning
were reduced. This suggests the potential of interventions for these
families in developing their social support and practical coping mechanisms.

Not surprisingly family relationships emerge as an important resource affecting
all members. Strong and direct associations were found between satisfaction
with life scores and positive marital and parent-child relationships. Like
any child, those children with Down syndrome in cohesive and harmonious
families were also less likely to have behaviour problems and more likely
to have higher levels of functioning.

Mothers expressing poor relationships with the child and family were more
likely to have high stress scores. Poor child relationships were likely
when the child with Down syndrome had a difficult temperament - high excitability -
and these children were also more likely to be in the lower IQ range and,
over time, to develop more behaviour problems. Mothers were also more likely
to express family problems if they were experiencing financial problems.
No other variable was significant. Thus early assessment indicating
difficult temperament and low ability in the child should led to interventions
aimed at establishing a more positive parent-child relationship. When
financial problems are apparent, specific financial support and advice on
money management may also avoid the likelihood of deterioration in relationships.
For these families help aimed at these issues would appear to be more
cost-effective than child centred interventions helping to promote quicker
development. Indeed it is these families who are more likely to be
unable to comply with the demands of many early intervention programmes
(Cunningham 1985).

The third aspect that emerged was the use of coping strategies.

Fathers' use of stoicism when appraising child problems was significantly
associated with higher satisfaction with life scores in mothers. It appeared
that those fathers who made light of the situation and refused to take it
too seriously provided some support for mothers who tended to have a more
emotional reaction. Clearly there is a sensitive balance in the interrelationships
of parents and one can imagine conflict resulting from fathers who persistently
ignore or make light of serious problems effecting the mother.

The strongest coping strategy to emerge was wishful thinking. When used
to cope with child problems there was a significant negative effect on the
well-being of mothers and fathers. It is not an effective strategy because
it is unlikely to resolve the problem which remains as a stressor. In a
proportion of the families wishful thinking remained dominant over the five
year period from childhood to teenage. However there was an indication that
it became less influential with an increase in passive acceptance of problems.
It is as if 'hope' that things will improve gives way to 'acceptance' and
the family increasingly adopts a routine of passive style of coping. This
is often seen with families of young adults with Down syndrome, even in
cohesive families who have worked hard and positively for their child. They
fall into easy, less stressful routines and the young person is incorporated
into the life-style of the parents. In such families the child and young
adult is also more likely to adopt a passive personal coping style, i.e.
learned helplessness.

These factors were also associated with parental external locus of control
i.e. feeling that one had little control over one's life. A link was also
found between neuroticism and wishful thinking. Parents with higher neuroticism
scores were more likely to adopt the strategy of wishful thinking, which
being ineffectual, leads to distress. A recent review (Knussen
and Sloper 1992) of studies using the resource and coping strategy model
outlined earlier, concluded that families of children with learning difficulties
most at risk had children with behaviour problems, reduced resources in
finances and materials (e.g. cars, washing machines etc.); inadequate social
support and personality and belief systems (e.g. neuroticism, low self-esteem);
were more likely to use fewer problem-solving and more passive emotion-focused
ways of coping, particularly wishful thinking.

In contrast we found that families who used problem solving strategies appeared
to score more highly on variables associated with well-being. Moreover,
such strategies were associated with higher functioning in the child with
Down syndrome.

Thus there is good support for the idea that interventions should focus
on helping those families at risk for passive coping mechanisms who are
likely to show poor psychological resources, external locus of control and
possible neuroticism. Zeitlin et al (1986,
1987) have described such an approach and its application to a small scale
intervention. Recently Kirkham (1993) demonstrated that mothers of young
children with disability who received such intervention improved in their
coping and communications and reported less depression and higher satisfaction
with life than a control group. This persisted over a two year period following
the training programme.

Although such approaches need to be used in the early years this type of
support may need to be offered in later years especially for those families
who may be gradually developing more passive and less expectant approaches.

Siblings

Research on siblings of children with disabilities presents a confused picture
with conflicting results often due to methodological problems.

A second approach has tried to investigate the roles and responsibilities
of siblings possibly associated with parental demands and higher expectations
due to some form of compensatory mechanism. Again the studies are equivocal.
McHale and Gamble (1989) found that siblings report more caregiving than
comparison children but Cuskelly and Gunn (1993) found no differences. No
support was found for the suggestion that parents have higher expectations
of the non-disabled child.

In the 1991 study, there were 63 siblings living at home and over 10 years
in our cohort. They were interviewed about their views of having a brother
or sister with Down syndrome, their roles and responsibilities and relationships
with the family. They completed self report questionnaires on self-worth,
anxiety and their perceived support and regard from their parents. Mothers
completed a scale of sibling behavioural problems. The analysis examined
these measures in combination with the measures on family functioning and
characteristics of the child with Down syndrome.

The main finding was of positive adaptation. Around 80% of the siblings
stated they had a positive relationships with their parents and their sibling
with Down syndrome and a positive or neutral effect of having a brother
or sister with Down syndrome. They also had positive perceptions of their
own self-worth. 95% were actively involved in helping roles with their sibling.
There was no indication of any detrimental effect and, in fact, the helping
role was more strongly associated with positive relationships with their
sibling with Down syndrome. Sixty percent felt they did not take on more
household responsibility than their peers whilst 21% felt they did more
and 19% felt they did the same amount.

As with our previous results the majority of mothers felt that having a
brother or sister with Down syndrome had had a positive effect on their
other children. This was seen in achievements and increased pro-social behaviour.
Again, many mothers felt that the care needs of the child with Down syndrome
had reduced the opportunities for family leisure time and the siblings own
time for themselves.

A complex picture emerged for 20% showing signs of poorer adaptation. No
effect was found for age or older sisters being at risk. Eighteen percent
of the siblings were rated with above threshold on behaviour problems but
this was not significantly related to any characteristic of the child with
Down syndrome. As found in our earlier studies the main factor associated
with behaviour problems was family relationships. Within this all variables
associated with mothers' adaptation but not fathers' adaptation were significant.
This highlights the significant role mothers play in fostering family relationships.
There was an indication of lower levels of behaviour problems in siblings
for those fathers who used social support as a coping mechanism to deal
with problems associated with their child with Down syndrome. Thus it appears
that the behaviour problems of the child with Down syndrome is not the main
factor affecting sibling problems. Of course it may be that where the behavioural
problems of the child with Down syndrome effect family relationships and
particularly mothers adaptation and coping this in turn influenced family
relationships and then sibling behaviour. But the siblings are not at
risk if there are sufficient resources to cope with such behaviour - for
example the use of the social networks and support by both parents. They
take their view of the sibling with Down syndrome from their parents.

The minority of siblings with low self-worth scores had brothers and sisters
with Down syndrome who had lower self-sufficiency scores and so placed higher
demands on family resources and restrictions on activities. These siblings
were also more likely to have higher trait anxiety scores and lower perceived
support and regard from their parents. This may reflect problems in parental
expectations and demands upon the sibling. Some felt that their brother
or sister with Down syndrome was a burden on their own lives and these siblings
also had higher behaviour problems. Many were also more likely to have perceived
themselves as different to their peers in the amount of help they gave with
caretaking activities - either more or less. Most teenagers use social comparison
processes in the development of self-concept and self-worth. Conformity
with the peer group is an important part of this. Thus these siblings might
perceive that their families were different and, by association, they were
different.

It would appear, therefore, that there is a small group of siblings at risk,
especially if the child with Down syndrome has low self-sufficiency skills
thus requiring more caretaking and supervision, are of an anxious disposition,
feel they receive low levels of parental support and encouragement, the
sibling with Down syndrome is a burden and they or their families are different.
Within this complex there are numerous explanatory mechanisms. The risk
to these siblings is reduced when families have the resources to balance
the needs of the all members and take a positive view of each other. For
those in families that are somewhat insular and isolated, possibly due to
the demands of the less independent child with Down syndrome, there is a
risk that they perceive themselves as different and develop lower self-worth.
Mothers are strongly influential but there are indications that fathers
can play a significant role to counter act the demands placed on mothers
of the child with Down syndrome and this is more likely when fathers use
their social support network. Clearly these issues require more thorough
investigation before we can develop interventions.

Factors associated with the development of
the child with Down syndrome

I will describe the factors associated with the outcome measures of developmental
age and mental ability, attainments (academic and self-sufficiency), behaviour
problems and social life.

Mental ability and early development:

In the first three years of life children with severe health (mainly heart)
problems made slower physical progress but no effect was found for mental
ability. Early intervention and intense structured training during the first
two years of life had an immediate small impact on the targeted behaviours
but no generalised or long term effects and the main predictor of later
scores was earlier scores on the same test - although this was modest in
the first year or so. These findings are similar to those reported in other
studies (e.g.
Gibson
and Field, 1988;
Shonkoff et al., 1992).

From around two years of age - mental ages of around 18 -24 months - significant
associations appeared between mental age scores and social class and educational
level of parents - with the latter showing the strongest influence. By 4
to 5 years the girls had higher mean group scores than the boys. This is
the same pattern as is found for ordinary children.

From around the second to third year of life the mental ability scores on
the children became increasing stable with correlations falling in the 0.7
to 0.9 range. Carr (1988) reports similar levels with a correlation of 0.9
between IQs measured in the early years and those at 21 years and good prediction
for around 80% of the sample. She confirmed a rise in mental ability scores
in the later teenage and early adult years except for the young people with
Down syndrome who were most severely disabled.

Attainments:

Academic attainments( reading, writing, number skills) and self-sufficiency
scores have steadily increased over the years for over 90% of the children.
The most powerful predictor of progress has been the child's mental ability
score accounting for around 60% of the variance for academic attainments
and 40 to 50 % for self-sufficiency scores.

For academic attainments greater progress was consistently and independently
found for those children attending mainstream schools and who had high attentional-low
distractibility scores. In 1986 we also found girls to have higher attainments
than boys but this was reducing in significance by 1991. However girls were
found to be less distractible than boys. In 1986 we found that children,
and more likely boys, of fathers with low locus of control scores made less
progress. Thus fathers who feel they may not be able to exert much influence
on events may be less inclined to become actively engaged in the child's
education or have different aspirations. In 1991 more progress was found
for children whose mothers used more practical and problem solving coping
strategies to deal with child-related problems.

An indirect influence was found for occupation and educational level. Children
of non-manual and higher educated parents were more likely to be placed
in mainstream schools from the early years. We found that about half of
the cohort children went to a mainstream pre-school setting. This was significantly
more than a comparison group who had no early intervention support at that
time. However the effect was due to more lower educated manual occupation
families seeking mainstream school from our cohort. Thus our supportive
intervention appeared to help these families to be more aware and assertive.
By the teenage years only 10% of the cohort remained in mainstream school
and 90% of these children resided in non-manual families.

We also found that the main factors related to attendance at mainstream
school was mental ability and low distractibility scores. Yet of the children
in special schools 15% had similar levels of ability and 63% similar levels
of attention, and although not statistically significant, more of this group
came from manual families. Thus one can speculate that families attitudes
towards education, and strategies and skills for dealing with professionals
are directly influential on educational placement and thus have an effect
on academic attainment.

Self-sufficiency scores are more influenced by family factors than academic
attainment. In 1986 we found that once we controlled for mental age, higher
self-sufficiency scores were associated with children whose mothers used
practical and problem solving coping strategies and lower for those who
tended to use wishful thinking. Children who had fewer behaviour problems
and were less excitable also gained higher scores. However the effect of
mothers style of coping was still significant even for children with difficult
temperaments and behaviour.

Environmental influences appear to have a greater impact on the more able
children. For example, the fifteen children with mental ages under 32 months
in 1986 made half the progress in self-sufficiency than the rest of the
sample by 1991. Furthermore mental age only accounted for 21% of the variance
on self-sufficiency progress for the 85% more advanced children compared
to 44% for the whole sample.

Thus, for the majority of children, as they got older the link between
cognitive ability and development of life skills became weaker and the influence
of family factors correspondingly stronger. In other words, although their
cognitive ability imposes limitations on their intellectual achievements,
by the time they reach mid to late childhood (with mental ages over 3-4
years) most have the ability to learn a wide range of life skills and will
do so when supported by their family and given applicable educational opportunities.

Behaviour problems

The level of behaviour and management problems decreased over the years.
However most of the children who demonstrated persistent behaviour problems
from early/mid-childhood still had problems in the teenage years and need
high levels of supervision. Up to the late childhood years the children
rated as having behaviour problems were more likely to have low mental abilities,
high excitability scores, a higher incidence of respiratory infections and
low self-sufficiency scores. As noted earlier, when these factors were controlled
for, this group also had poorer scores on family cohesion and parent-child
relationships, inadequate housing - which reflected financial problems,
low social support for parents and unemployment in fathers, mothers using
passive coping strategies and those with high neuroticism scores and finally
strain from current life events. It is difficult to determine cause and
effect in these factors but parent coping styles, financial problems and
recurrent child health are likely to effect the management style and interrelationships
in the families. They are also reasonable targets for intervention. By 1991
the main predictor of behaviour problems was the ratings in 1986. This
suggests that the time for preventative interventions with these families
is in the early years.

Social life

Social life was measured in terms of the children's contact with organised
activities e.g. clubs, teams and informal activities e.g. contacts with
friends, other people, going on family outings. Although higher overall
scores were more likely for the more able children with fewer behaviour
problems several family characteristics were far more influential on the
type of social life they had. Thus children in families with an active-recreational
orientation, younger mothers and more siblings had a wider social life.

These two aspects, organizational contacts and informal contacts, were not
necessarily related. Some children had no friends but attended clubs and
others had many informal contacts but did not attend organised activities.
They were also predicted by different factors. The children with higher
levels of informal contacts had younger mothers who used practical coping
strategies and did not have financial problems. Children who attended no
organised activities were from families with poor marital relationships
and fathers with personality problems and stress. In contrast if the child
attended a mainstream school they were more likely to engage in a wider
range of social activities.

Thus the results identify many environmental factors related to family
functioning which have direct impact on the development, behaviour and social
life of the child with Down syndrome.

Conclusions

The overall impression of the families and children with Down syndrome is
one of normality. The factors that influence their well being and that of
the child are largely the same as those influencing any child and family.
The research has emphasised the wide range of individual differences between
and within the families and between the children themselves. Consequently
generalised statements and assumptions based on the fact that Down syndrome
is present should be avoided.

It cannot be assumed that the family or individual members are 'at risk'
for psychological problems just because one member has Down syndrome. We
found that the majority of families (60-70%) in the cohort were harmonious
with high levels of family cohesion and perceived satisfaction with life
and relatively normal levels of stress. They had adapted positively to their
child with Down syndrome and report no persistent negative psychological
effects on their lives or their other children. In fact they were more likely
to feel the child had positively contributed to the family.

I do not wish to minimise the problems these families face. They do experience
considerable trauma following the birth and, in their role as parents or
relatives of a child with a major disability, have to develop new knowledge
and skills and re-construct their ideas about themselves, their values and
aspirations. They often face a society with little understanding and much
prejudice. They have to learn to deal with a wide range of professionals
and agencies and develop assertive negotiating skills in order to obtain
the best resources for their child. They also have to develop practical
problem solving strategies and maintain a positive expectant attitude for
their child despite the many set-backs, discouragement and slow progress.
The fact that so many families cope well is a testimony to their commitment
to their child, and their adaptability and strengths, rather than a lack
of problems.

They do need accurate information and quality support from services about
their child's needs and available services. They need this at critical points
in the life-cycle of their child. Certainly in the first months, at the
time they select pre-school and schools, transitions from childhood to puberty,
teenager to young adult, school to college and independent living.

About a third of the families experience difficulties reflected in higher
stress and lower satisfaction with life. This was associated with the child
characteristics of lower mental ability, behaviour problems, excitable temperament
and recurring infections. It was also associated with a cluster of family
characteristics e.g. lower levels of utilitarian resources associated with
money, housing and employment; lower levels of social support, health problems
of parents, and psychological resources such as personality and use of ineffectual
coping strategies. All of these factors were found to influence the development
and behaviour of the children. They also mediated the levels of stress and
satisfaction with life experienced by the parents. Several of these factors
are likely to be influenced by intervention.

Of importance was the finding that the best predictor of any measure e.g.
stress in the parent, mental ability, self-sufficiency, behaviour problems
in the child was the score on the same measure in the previous study 5 years
and 9 years earlier. This strongly suggests the need for intervention in
the earlier years.

The research clearly indicates that such intervention should be focused
on the family as a unit and not the child or merely the pathology of the
child. Generalised programmes of therapy and training which aim to counteract
the potential development delay or deficits associated with the child's
intellectual disability have not proved to be that effective. This is not
surprising given the vast amount of individual differences in the children
with Down syndrome and their families. Hence programmes which are automatically
applied to all children with Down syndrome are simplistic and not efficient.

My approach is to enter into a partnership relationship with the family
in which I explain much of the above research and the model I use to assess
their needs. I start by exploring the likely demands on the family resources
of the child, relate this to the resources they have available and their
strategies of coping. The three sources of demand are:

1) the caretaking demands - these concern the level of
personal self-sufficiency in the child (e.g. feeding, dressing bathing,
toileting), ill health, mobility. The extent to which the family cope with
such demands depends largely on their utilitarian resources, practical social
support and health/energy resources. Thus the needs will relate to finance,
housing, transport, laundry, home help, respite care, special diets, labour
saving equipment etc. In some families these demands will be influenced
by belief systems e.g. the extent to which they encourage self-sufficiency
in the child and practical problem solving ability in seeking help and advice.

2) supervisory demands - such as length of time the child
can be left alone, whether the child can be trusted in the home, with friends
or when out on excursions. These are related to the child's behaviour and
ease of management. They are more frequently associated with psychological
resources underlying parental values about child behaviour and their ability
to apply appropriate and consistent management procedures. However they
also encompass social support, housing, and health. Thus the family needs
include respite care, professional help with development and applying behavioural
management skills. Often, it is essential to consider parental attitudes
and their beliefs that they can be effective. Thus programmes supporting
their confidence in their parenting skills are important.

3) affective demands - some parents experience low feelings
of fulfilment in their parental role. They find it difficult to have positive
feelings towards the child. This is sometimes due to the lack of affective
behaviours and responses from the child and/or the child not fulfilling
the parents hopes and expectations of a child. Such feelings are very common
following the disclosure of the diagnosis. At this time the family needs
supportive counselling and help in learning how to observe their child and
understand and interpret the child's behaviour and development. They may
also need to reflect on their feelings about disability and their changed
role and expectations of parenthood. These can change and fluctuate as the
child grows and the emerging person is observed. They can change as a result
of other family factors and changed circumstances. The adaptation of family
members (mothers, fathers, siblings, grandparents or significant others)
is individual. Support therefore, must be focused on their individual needs
but nested within a detailed understanding of their family based upon a
careful assessment of resources and the coping mechanisms adopted by members
when faced with problems.

We will only be able to predict the full potential of people with Down syndrome
when they and their families have access to this type of support at key
points in their life-cycle.

Acknowledgements

Based on a paper presented at the International Conference-'People with
Down Syndrome on The Threshold of the 21st Century', Rome 23-25 October
1996.

I am grateful to all who have worked on the Manchester cohort since 1973,
particularly Dr Trica Sloper and Steve Turner who were responsible for the
more recent work.

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Gath, A. (1973). The school age siblings
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